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NETS1HD study: development of a Hirschsprung’s disease core outcome set
  1. Benjamin Saul Raywood Allin1,
  2. Timothy Bradnock2,
  3. Simon Kenny3,
  4. Jennifer J Kurinczuk1,
  5. Gregor Walker2,
  6. Marian Knight1
  7. The NETS1HD Collaboration
  1. 1 National Perinatal Epidemiology Unit, University of Oxford, Headington, UK
  2. 2 Royal Hospital for Children, Glasgow, UK
  3. 3 Alder Hey Children’s Hospital, Liverpool, UK
  1. Correspondence to Dr Benjamin Saul Raywood Allin, National Perinatal Epidemiology Unit, University of Oxford, Richard Doll Building, Old Road Campus, Headington, Oxford OX3 7LF, UK; benjaminallin{at}


Objective The objective of this study was to develop a Hirschsprung’s disease (HD) core outcome set (COS).

Methods Candidate outcomes were identified from a systematic review and stakeholder nomination. A three-phase Delphi process and consensus meeting were used to prioritise candidate outcomes based on scores assigned by stakeholder participants using a nine-point scale. In phases two and three, participants were shown graphical representations of their panel’s scores and all panels’ scores respectively for each outcome from the previous phase. After the third phase, outcomes prioritised by two or three panels were taken forward to the consensus meeting. The COS was formed from the 10 highest scoring outcomes meeting the threshold for inclusion (≥70% 7–9 and <15% 1–3).

Results Eighty-nine stakeholders (82%) completed all three phases of the Delphi process. Seventy-four outcomes were assessed in phase one of the Delphi process, the following 10 of which met criteria for inclusion in the COS: (1) death with cause specified, (2) long-term faecal incontinence, (3) long-term voluntary bowel movements without need for enemas, or rectal or colonic irrigation, (4) long-term psychological stress for the individual with Hirschsprung’s disease, (5) long-term urinary incontinence, (6) objective score of quality of life, (7) objective score of bowel function, (8) unplanned reoperation, (9) >need for a permanent stoma, (10) enterocolitis.

Conclusions This HD COS is formed of 10 outcomes deemed important by key stakeholders. Use of this COS in research will reduce outcome reporting heterogeneity and increase our ability to identify gold standard treatments for HD.

  • Hirschsprung’s Disease
  • Core Outcome Set
  • Paediatric Surgery
  • Gastroenterology

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  • GW and MK contributed equally.

  • Contributors BSRA was responsible for design of the study, day-to-day management of the study, analysis of data and drafting of the manuscript. TB, SK, GW, JJK and MK were responsible for design of the study, management of the study and contributed to critical revisions of the manuscript. Members of NETS1HD contributed critically to the academic content of the study and have reviewed and critically revised the manuscript.

  • Funding MK is funded by a National Institute for Health Research (NIHR) Professorship. BSRA is funded by an NIHR Doctoral Research Fellowship.

  • Disclaimer The views expressed are those of the author(s) and not necessarily those of the NHS, the NIHR or the Department of Health. The NIHR had no role in design and conduct of the study; collection, management, analysis and interpretation of the data; and preparation, review or approval of the manuscript; and decision to submit the manuscript for publication.

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Collaborators The members involved in NETS1HD Collaboration who went through completion of all three phases of the Delphi process contributed significantly to the development of this HD core outcome set: Martin Ward Platt, Sridhar Ramaiah, Nim Subhedar, Anna-May Long, Mike Stanton, David Burge, Kokila Lakhoo, Stefano Giuliani, Paul Johnson, Silke Wagener, Graham Lamont, Nigel Hall, Isaac Philip, Richard Lindley, Catherine Bradshaw, Ian Sugarman, Simon Clarke, Claire Clark, Clare Rees, Ivo de Blaauw, Bala Eradi, Janne Suominen, Mark Tighe, Andrew Barclay, Diana Flynn, Sara Jones, Keith Hipkin, Kelly Arnold, Clare Lambert, Tracey Fuller, Stella Taylor, Patricia Umney, Lacey Edwards, Poly The, Michelle Luckett, Claire Ryder, Jennie Kennerley, Elizabeth Faulkner, Claire Radtke, Holly Stowell, Lucy Somers, Rachel Tabone, Toni Colgan, Helen Sanderson, Lorina Woodward, Raeley Grant, Helen Fowler, Cynthia Rauscher, Marianne Woller, Katrina Randon, Katherine Brandist, Vanessa Riordan, Vicky Taylor, Nicola Purdy, Lorraine Pearce, Kylee Murray, Lisa Coleman, Lucy Nall, Lisa McIntosh, Nicola Krijger, Macaley Smith, Sharon Magalona, Carolyn Walker, Alison Broadbent, Stephen Caldwell, Christeen Smith, Ali Wright, Claire Bohr, David Pambakian, Geoffrey Piper, Neil Woller, Katherine Woller, Osvaldo Borrelli and Marion Lambourne. Based on their attendance at the consensus meeting, the following people form the NETS1HD collaboration: Sarah Almond, Rebecca Craven, Karen Dick, Richard England, Alan Fenton, Susan Grieve, Jonathan Hind, Vicky Hogg, Gemma Jordan, Victoria Lane, Marc Levitt, Gerry McGregor, Alison Sharrard, Jonathan Sutcliffe and Katie Yearsley.

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